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Case Reports in Gastrointestinal Medicine

Volume 2019, Article ID 2762389, 3 pages
https://doi.org/10.1155/2019/2762389

Case Report
Severe Hyperbilirubinemia: A Rare Complication of Lyme Disease

Maarij Baig,1 Lin Zheng ,2 and Alka Farmer2

1
Internal Medicine Residency Program, Inspira Medical Center in Vineland, Vineland, NJ 08360, USA
2
Division of Hospital Medicine, Department of Medicine, Cooper Medical School of Rowan University, Camden, NJ 08103, USA

Correspondence should be addressed to Lin Zheng; zheng-lin@cooperhealth.edu

Received 13 October 2019; Accepted 5 December 2019; Published 28 December 2019

Academic Editor: Engin Altintas

Copyright © 2019 Maarij Baig et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Gastrointestinal signs and symptoms are common in the early stages of Lyme disease. However, hyperbilirubinemia from Lyme
disease is extremely uncommon. There are only two case reports in literature attributing Lyme disease to hyperbilirubinemia. Here
we report a case of severe hyperbilirubinemia as the presenting symptom of Lyme disease. Other plausible etiologies have been ruled
out after extensive workups, including liver biopsy. His hyperbilirubinemia gradually resolved after being started on doxycycline.
With high incidence of Lyme disease, it should be considered for patients who present with hyperbilirubinemia in endemic areas
with possible tick exposure.

1. Introduction
Lyme disease is a multisystemic infection caused by the tick-
borne spirochete, Borrelia burgdorferi. Gastrointestinal
involvement, such as acute hepatitis, could present in the early
stages of Lyme disease [1]. However, hyperbilirubinemia
secondary to Lyme disease is extremely uncommon. There are
only two case reports in literature attributing Lyme disease to
hyperbilirubinemia; one of which was in a pediatric patient
[2, 3]. Here we present a rare case of hyperbilirubinemia
secondary to Lyme disease.
2. Case Presentation
A 23-year-old male presented to our hospital with jaundice,
fever, and arthralgia in early September. He had been in his
usual state of health until approximately six days prior to his
presentation. He had experienced diffuse arthralgia and a tem-
perature of 38.9°C. He was also told he had yellowing of his
eyes and skin, which prompted his visit to the Emergency
Department. The patient had a past history of splenectomy
five years prior to presentation resulting from a motor vehicle
accident. Most recently, the patient states he had discontinued
his intravenous heroin and moved from a tent in a rural town
into a group home and restarted Suboxone.
On the day of presentation, initial vitals showed a
temperature of 36.7°C, blood pressure of 102/56 mmHg, heart
rate of 105 beats per minute, respiratory rate of 20 breaths per
minute, and oxygen saturation of 98% on room air. On physical
exam, the patient was severely jaundiced with scleral icterus.
Examination of the abdomen, joints, skin, and lymph nodes
was unremarkable and there were no stigmata of chronic liver
disease. His initial laboratory tests showed WBC 25.6 × 103/µL,
creatinine of 4.5 mg/dL, total bilirubin 11.4 mg/dL (direct
9.8 mg/dL, indirect 1.6 mg/dL) (Figures 1 and 2), Alanine
aminotransferase (ALT) of 46 U/L (Unit/Liter), aspartate
aminotransferase (AST) of 59 U/L, and Alkaline Phosphatase
(ALK) of 127 U/L (Figure 3).
Abdominal ultrasound was negative, while CT of the
thorax, abdomen, and pelvis showed diffuse pulmonary
infiltrates, extensive parenchymal changes bilaterally in the
lung bases with mediastinal lymph nodes and mesenteric
portal hepatic adenopathy. He was started on intravenous
piperacillin-tazobactam for possible pulmonary infection.
Extensive workup for conjugated hyperbilirubinemia
included viral hepatitis screen, 𝛼 antitrypsin antibody (A1AT),
ceruloplasmin, CMV, HIV, antinuclear antibody (ANA), and
antismooth muscle antibody, all of which were negative. His
LDH was 439 U/L (normal).
There was the concern for tick-borne disease because the
patient lived in a tent in the Mid-Atlantic region. Peripheral
blood smears were negative for any parasite. Serum screening
tests for Lyme disease, Babesiosis, and Ehrlichiosis were
ordered. He was started on empirical doxycycline on the third
hospital day.
2 Case Reports in Gastrointestinal Medicine

0
Day 1 Day 2 Day 3 Day 4 Day 5 Day 6 Day 7 Day 12 Day 13 Day 14
Creatinine

Figure 1: Serum creatine level (in mg/dL) by the hospital day.

25

20

15

10

0
Day 1 Day 3 Day 4 Day 7 Day 13
Total bili
Direct bili

Figure 2: Total and direct bilirubin level (mg/dL) by the hospital day.

70

60

50

40

30

20

10

0
Day 1 Day 2 Day 3 Day 4 Day 6 Day 7
AST
ALT

Figure 3: Alanine aminotransferase (ALT, in U/L) and aspartate aminotransferase (AST, in U/L) by the hospital day.
Case Reports in Gastrointestinal Medicine 3

His acute renal injury peaked on hospital day 3 (creatinine including acute renal failure, which could be attributed to
7.3 mg/dL) then gradually trended down. Creatinine improved Lyme disease associated glomerulonephritis [6].
to 3.0 mg/dL on hospital day 7 (Figure 1). Stool studies, includ- In conclusion, although highly uncommon, hyperbiliru-
ing Escherichia coli 0157:H7, were negative. binemia may be the presenting sign of Lyme disease. Lyme
His total bilirubin continued to rise to 20.6 mg/dL (direct disease should be considered for patients from endemic areas
19.3 mg/dL) on the 7th hospital day (Figure 2), His WBC with possible tick exposure.
remained elevated at 20 × 103/µL. However, his AST/ALT
remained normal (Figure 3). His International Normalization
Ratio (INR) was normal throughout his hospitalization. Conflicts of Interest
The patient remained afebrile and normotensive through-
out his stay. Magnetic resonance cholangiopancreatography The authors declare that they have no conflicts of interest.
(MRCP) was subsequently performed and showed no evidence
of obstruction or intrahepatic dilatation. Repeat US was per-
References
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elevated liver function tests among Lyme disease patients [4].
However, hyperbilirubinemia is highly uncommon. Abnormal
bilirubin was only seen in 3 out of 115 cases in this study [4].
Similarly, no single case of hyperbilirubinemia was reported
in another study, in which a total 314 patients with Lyme
disease were included [5]. To the best of our knowledge, this
is the first case of hyperbilirubinemia (20 mg/dL) of this
extreme attributed to Lyme disease.
Although highly uncommon, Lyme disease should be con-
sidered in the differential diagnosis of hyperbilirubinemia,
particularly in patients who are at risk of severe infection and
end organ damage and are living in an endemic area or have
recently travelled to an endemic area, regardless of the pres-
ence of rash. Our patient lived in a tent in a wooded area in
the Mid-Atlantic region. He presented to the hospital with
fever in September. These risk factors should prompt the
workup for tick-borne disease.
The combination of ELISA to detect IgM and IgG anti-B.
Burgdorferi antibodies and Western blot provides the greatest
sensitivity and specificity for the laboratory diagnosis of Lyme
disease. Our patient confirmed for Borrelia burgdorferi infec-
tion. He responded to doxycycline treatment. Extensive
workups for other causes of jaundice, including liver biopsy,
were negative. He also had multiple end organ damage,
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